| Summary: | Idiopathic orbital inflammation (IOI) is a noninfectious inflammatory disease whose etiology remains unknown. Treatment is focused on reducing inflammation, which becomes challenging in nonresponding cases. We report the case of a 59-year-old woman with refractory IOI that showed a positive response to tocilizumab therapy. The patient was diagnosed with a unilateral sclerosing IOI for 9 years and showed a negative control with previous oral steroids, peribulbar steroid injections, radiotherapy, immunosuppressors, and intravenous rituximab. After the initiation of 8 mg/kg intravenous tocilizumab, a complete reduction of the pain and the orbital inflammation signs was observed and her condition remained stable for the following 6 years under a monthly dose of 4 mg/kg. In recalcitrant IOI cases, tocilizumab could be considered a possible treatment to reduce inflammatory signs and symptoms with positive long-term outcomes as in our case.
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